This current case is the first report of severe unilateral (left-sided) post-traumatic trigeminal neuropathic pain involving all branches of the trigeminal nerve after custom-made total temporomandibular joint replacement (TMJR). After a bilateral TMJR procedure a, 43-year old female patient complained of left-sided lingual and mental nerve hypoesthesia. In the following months, the patient developed unsustainable neuropathic pain at the area of cutaneous distribution of the mental (V3) and infraorbital (V2) nerves combined with hypoesthesia in all trigeminal divisions. Neurosensory deficits in V1-3 were confirmed by quantitative sensory testing and electrophysiological studies. Treatments consisted of anti-depressants, anti-epileptics, opioids and eventually radiofrequent ablation of the peripheral branches without notable improvement in pain complaints or sensory deficits. The current case warrants temporomandibular joint surgeons for the possible risk of post-traumatic trigeminal neuropathic pain. •Trigeminal neuropathic pain may follow temporomandibular joint replacement.•Symptoms may spread far beyond the affected dermatome on the basis of an inflammatory and/or excitotoxic response.•Quality of life deteriorates with persistent neuropathic pain.•Conservative treatment is disappointing.•A multidisciplinary approach with thorough investigation is advised.